How the enzyme ADAMTS9 affects tiny hair-like cilia in developing and diseased kidneys

Extracellular protease modulation of the cilium transition zone in kidney development and disease

NIH-funded research Univ of Massachusetts Med Sch Worcester · NIH-11251199

This project looks at how the protein ADAMTS9 alters cilia on kidney cells and how that may lead to childhood medullary cystic kidney disease (nephronophthisis).

Quick facts

Grant typeR01 grant
Study typeNIH-funded research
Funding institutionUniv of Massachusetts Med Sch Worcester NIH-funded
Lab location1 site (Worcester, United States)
Project IDNIH-11251199 on NIH RePORTER

What this research studies

Researchers are working to understand how ADAMTS9, a secreted enzyme, helps build the primary cilium — a tiny hair-like structure important for kidney-cell function — and why loss of this enzyme leads to nephronophthisis. They discovered that ADAMTS9 cuts a cilia-related protein called TMEM67 and will test how that cleavage affects cilium assembly and cell signaling. The team will use genetically engineered mice that lack Adamts9 in the kidney and perform 'rescue' experiments in human cell models while tracking protein fragments with N-terminomics. Although most work is in the lab and in mice, the results could point to molecular targets for future patient tests or therapies.

Who could benefit from this research

Good fit: People (often children or families) affected by autosomal recessive nephronophthisis or who have known ADAMTS9 or TMEM67 mutations would be the most relevant candidates to follow or contribute to related future studies.

Not a fit: Patients with kidney disease caused by unrelated mechanisms (for example diabetic kidney disease) or without cilia-related gene mutations are less likely to benefit directly from this specific work.

Why it matters

Potential benefit: If successful, this work could reveal a new molecular step that causes nephronophthisis and point to targets for future treatments or diagnostic tests.

How similar studies have performed: Prior animal and cell studies have shown ADAMTS9 is required for ciliogenesis and that TMEM67 mutations cause similar kidney disease, but the idea that ADAMTS9 cleaves TMEM67 as a key step is a new finding.

Where this research is happening

Worcester, United States

Researchers

About this research

  1. This is an active NIH-funded research project — typically early-stage science, not a clinical trial accepting patient enrollment.
  2. Some NIH-funded labs run parallel clinical studies or seek volunteers for related work. To check, contact the principal investigator or institution listed above.
  3. For full project details, budget, and progress reports, visit the official NIH RePORTER page below.
Conditions Autosomal Recessive Medullary Cystic Disease
Last reviewed 2026-06-13 by the Find a Trial editorial team. Information on this page is for educational purposes and is not medical advice. Always consult qualified healthcare professionals about clinical trial participation.